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Select item 112240281.

Hepatic oxylipin profiles in mouse models of Wilson disease: New insights into early hepatic manifestations

Tagreed A. Mazi, Noreene M. Shibata, Gaurav V. Sarode, Valentina Medici

Biochim Biophys Acta Mol Cell Biol Lipids. Author manuscript; available in PMC 2024 Jul 5.

Published in final edited form as: Biochim Biophys Acta Mol Cell Biol Lipids. 2024 Mar; 1869(2): 159446. Published online 2023 Dec 9. doi: 10.1016/j.bbalip.2023.159446

PMCID:: PMC11224028

Abstract Article PDF–2.2M

Select item 107212602.

Genome editing without nucleases confers proliferative advantage to edited hepatocytes and corrects Wilson disease

Agnese Padula, Michele Spinelli, Edoardo Nusco, Xabier Bujanda Cundin, Filomena Capolongo, Severo Campione, Claudia Perna, Amy Bastille, Megan Ericson, Chih-Chieh Wang, Shengwen Zhang, Angela Amoresano, Mariana Nacht, Pasquale Piccolo

JCI Insight. 2023 Nov 8; 8(21): e171281. Published online 2023 Nov 8. doi: 10.1172/jci.insight.171281

PMCID:: PMC10721260

Abstract Article PDF–2.6M

Select item 98701413.

Atp7b-dependent choroid plexus dysfunction causes transient copper deficit and metabolic changes in the developing mouse brain

Clorissa L. Washington-Hughes, Shubhrajit Roy, Herana Kamal Seneviratne, Senthilkumar S. Karuppagounder, Yulemni Morel, Jace W. Jones, Alex Zak, Tong Xiao, Tatiana N. Boronina, Robert N. Cole, Namandjé N. Bumpus, Christopher J. Chang, Ted M. Dawson, Svetlana Lutsenko

PLoS Genet. 2023 Jan; 19(1): e1010558. Published online 2023 Jan 10. doi: 10.1371/journal.pgen.1010558

PMCID:: PMC9870141

Abstract Article PDF–3.5M

Select item 87590434.

Hepatic Steatosis in the Mouse Model of Wilson Disease Coincides with a Muted Inflammatory Response

Aline Gottlieb, Som Dev, Lauren DeVine, Kathleen L. Gabrielson, Robert N. Cole, James P. Hamilton, Svetlana Lutsenko

Am J Pathol. 2022 Jan; 192(1): 146–159. doi: 10.1016/j.ajpath.2021.09.010

PMCID:: PMC8759043

Abstract Article PDF–5.0M

Select item 83257785.

Copper Sulfide Facilitates Hepatobiliary Clearance of Gold Nanoparticles through the Copper-Transporting ATPase ATP7B

Xiaodong Wang, Liangran Guo, Sihang Zhang, Yuan Chen, Yi-Tzai Chen, Binbin Zheng, Jingwen Sun, Yuyi Qian, Yixin Chen, Bingfang Yan, Wei Lu

ACS Nano. Author manuscript; available in PMC 2021 Aug 1.

Published in final edited form as: ACS Nano. 2019 May 28; 13(5): 5720–5730. Published online 2019 Apr 18. doi: 10.1021/acsnano.9b01154

PMCID:: PMC8325778

Abstract Article PDF–1.4M

Select item 83132396.

Cell-Type-Specific Gene Expression in Developing Mouse Neocortex: Intermediate Progenitors Implicated in Axon Development

Francesco Bedogni, Robert F. Hevner

Front Mol Neurosci. 2021; 14: 686034. Published online 2021 Jul 12. doi: 10.3389/fnmol.2021.686034

PMCID:: PMC8313239

Abstract Article PDF–1.6M

Select item 80823937.

Comprehensive RNA-Seq Analysis of Potential Therapeutic Targets of Gan–Dou–Fu–Mu Decoction for Treatment of Wilson Disease Using a Toxic Milk Mouse Model

Taohua Wei, Wenjie Hao, Lulu Tang, Huan Wu, Shi Huang, Yue Yang, Nannan Qian, Jie Liu, Wenming Yang, Xianchun Duan

Front Pharmacol. 2021; 12: 622268. Published online 2021 Apr 15. doi: 10.3389/fphar.2021.622268

PMCID:: PMC8082393

Abstract Article PDF–6.0M

Select item 79525808.

Systemic deletion of Atp7b modifies the hepatocytes’ response to copper overload in the mouse models of Wilson disease

Abigael Muchenditsi, C. Conover Talbot, Jr., Aline Gottlieb, Haojun Yang, Byunghak Kang, Tatiana Boronina, Robert Cole, Li Wang, Som Dev, James P. Hamilton, Svetlana Lutsenko

Sci Rep. 2021; 11: 5659. Published online 2021 Mar 11. doi: 10.1038/s41598-021-84894-3

PMCID:: PMC7952580

Abstract Article PDF–10M

Select item 79387709.

BraInMap elucidates the macromolecular connectivity landscape of mammalian brain

Reza Pourhaghighi, Peter E. A. Ash, Sadhna Phanse, Florian Goebels, Lucas Z. M. Hu, Siwei Chen, Yingying Zhang, Shayne D. Wierbowski, Samantha Boudeau, Mohamed T. Moutaoufik, Ramy H. Malty, Edyta Malolepsza, Kalliopi Tsafou, Aparna Nathan, Graham Cromar, Hongbo Guo, Ali Al Abdullatif, Daniel J. Apicco, Lindsay A. Becker, Aaron D. Gitler, Stefan M. Pulst, Ahmed Youssef, Ryan Hekman, Pierre C. Havugimana, Carl A. White, Benjamin C. Blum, Antonia Ratti, Camron D. Bryant, John Parkinson, Kasper Lage, Mohan Babu, Haiyuan Yu, Gary D. Bader, Benjamin Wolozin, Andrew Emili

Cell Syst. Author manuscript; available in PMC 2021 Apr 22.

Published in final edited form as: Cell Syst. 2020 Apr 22; 10(4): 333–350.e14. doi: 10.1016/j.cels.2020.03.003

Correction in:: Cell Syst. 2020 Aug 26; 11(2): 208.

PMCID:: PMC7938770

Abstract Article PDF–2.0M

Select item 781385110.

Inhibiting NLRP3 inflammasome activation prevents copper-induced neuropathology in a murine model of Wilson’s disease

Jianjian Dong, Xun Wang, Chenchen Xu, Manli Gao, Shijing Wang, Jin Zhang, Haiyang Tong, Lulu Wang, Yongzhu Han, Nan Cheng, Yongsheng Han

Cell Death Dis. 2021 Jan; 12(1): 87. Published online 2021 Jan 18. doi: 10.1038/s41419-021-03397-1

PMCID:: PMC7813851

Abstract Article PDF–3.7M

Select item 699499011.

Metabolic dysregulation in the Atp7b^−/− Wilson’s disease mouse model

Clavia Ruth Wooton-Kee, Matthew Robertson, Ying Zhou, Bingning Dong, Zhen Sun, Kang Ho Kim, Hailan Liu, Yong Xu, Nagireddy Putluri, Pradip Saha, Cristian Coarfa, David D. Moore, Alli M. Nuotio-Antar

Proc Natl Acad Sci U S A. 2020 Jan 28; 117(4): 2076–2083. Published online 2020 Jan 10. doi: 10.1073/pnas.1914267117

PMCID:: PMC6994990

Abstract Article PDF–1.1M

Select item 645274412.

ATP7A delivers copper to the lysyl oxidase family of enzymes and promotes tumorigenesis and metastasis

Vinit Shanbhag, Kimberly Jasmer-McDonald, Sha Zhu, Adam L. Martin, Nikita Gudekar, Aslam Khan, Erik Ladomersky, Kamlendra Singh, Gary A. Weisman, Michael J. Petris

Proc Natl Acad Sci U S A. 2019 Apr 2; 116(14): 6836–6841. Published online 2019 Mar 19. doi: 10.1073/pnas.1817473116

PMCID:: PMC6452744

Abstract Article PDF–1.1M

Select item 631149813.

ATP7A and ATP7B copper transporters have distinct functions in the regulation of neuronal dopamine-β-hydroxylase

Katharina Schmidt, Martina Ralle, Thomas Schaffer, Samuel Jayakanthan, Bilal Bari, Abigael Muchenditsi, Svetlana Lutsenko

J Biol Chem. 2018 Dec 28; 293(52): 20085–20098. Published online 2018 Oct 19. doi: 10.1074/jbc.RA118.004889

PMCID:: PMC6311498

Abstract Article PDF–3.7M

Select item 621621114.

Epigenetic changes of the thioredoxin system in the tx-j mouse model and in patients with Wilson disease

Charles E Mordaunt, Noreene M Shibata, Dorothy A Kieffer, Anna Członkowska, Tomasz Litwin, Karl Heinz Weiss, Daniel N Gotthardt, Kristin Olson, Dongguang Wei, Stewart Cooper, Yu-Jui Yvonne Wan, Mohamed R Ali, Janine M LaSalle, Valentina Medici

Hum Mol Genet. 2018 Nov 15; 27(22): 3854–3869. Published online 2018 Jul 16. doi: 10.1093/hmg/ddy262

PMCID:: PMC6216211

Abstract Article PDF–32M

Select item 607503115.

DPM-1001 decreased copper levels and ameliorated deficits in a mouse model of Wilson's disease

Navasona Krishnan, Christy Felice, Keith Rivera, Darryl J. Pappin, Nicholas K. Tonks

Genes Dev. 2018 Jul 1; 32(13-14): 944–952. doi: 10.1101/gad.314658.118

PMCID:: PMC6075031

Abstract Article PDF–2.7M

Select item 553883616.

Targeted inactivation of copper transporter Atp7b in hepatocytes causes liver steatosis and obesity in mice

Abigael Muchenditsi, Haojun Yang, James P. Hamilton, Lahari Koganti, Franck Housseau, Lisa Aronov, Hongni Fan, Hannah Pierson, Ashima Bhattacharjee, Robert Murphy, Cynthia Sears, James Potter, Clavia R. Wooton-Kee, Svetlana Lutsenko

Am J Physiol Gastrointest Liver Physiol. 2017 Jul 1; 313(1): G39–G49. Published online 2017 Apr 20. doi: 10.1152/ajpgi.00312.2016

PMCID:: PMC5538836

Abstract Article

Select item 458828517.

Elevated copper impairs hepatic nuclear receptor function in Wilson’s disease

Clavia Ruth Wooton-Kee, Ajay K. Jain, Martin Wagner, Michael A. Grusak, Milton J. Finegold, Svetlana Lutsenko, David D. Moore

J Clin Invest. 2015 Sep 1; 125(9): 3449–3460. Published online 2015 Aug 4. doi: 10.1172/JCI78991

PMCID:: PMC4588285

Abstract Article PDF–1.3M

Select item 439519518.

Laser ablation inductively coupled plasma mass spectrometry imaging of metals in experimental and clinical Wilson's disease

Sorina Georgiana Boaru, Uta Merle, Ricarda Uerlings, Astrid Zimmermann, Christa Flechtenmacher, Claudia Willheim, Elisabeth Eder, Peter Ferenci, Wolfgang Stremmel, Ralf Weiskirchen

J Cell Mol Med. 2015 Apr; 19(4): 806–814. Published online 2015 Feb 20. doi: 10.1111/jcmm.12497

PMCID:: PMC4395195

Abstract Article PDF–1.1M

Select item 369582819.

A systems approach implicates nuclear receptor targeting in the Atp7b^−/− mouse model of Wilson’s Disease

Phillip Wilmarth, Kristopher Short, Oliver Fiehn, Svetlana Lutsenko, Larry David, Jason L. Burkhead

Metallomics. Author manuscript; available in PMC 2013 Jul 1.

Published in final edited form as: Metallomics. 2012 Jul; 4(7): 660–668. Published online 2012 May 8. doi: 10.1039/c2mt20017a

PMCID:: PMC3695828

Abstract Article PDF–1.9M

Select item 357241020.

A conditional knockout resource for the genome–wide study of mouse gene function

William C. Skarnes, Barry Rosen, Anthony P. West, Manousos Koutsourakis, Wendy Bushell, Vivek Iyer, Alejandro O. Mujica, Mark Thomas, Jennifer Harrow, Tony Cox, David Jackson, Jessica Severin, Patrick Biggs, Jun Fu, Michael Nefedov, Pieter J. de Jong, A. Francis Stewart, Allan Bradley

Nature. Author manuscript; available in PMC 2013 Feb 14.

Published in final edited form as: Nature. 2011 Jun 15; 474(7351): 337–342. Published online 2011 Jun 15. doi: 10.1038/nature10163

PMCID:: PMC3572410

Abstract Article PDF–1.1M

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